Unusual Presentation of Hemophagocytic Lymphohistiocytosis in a Kidney Transplant Patient

Yang, Danwen; Thamcharoen, Natanong; Marcus, Chelsea; Varkaris, Andreas; Aird, William C.; Khankin, Eliyahu V.; Cardarelli, Francesca

doi:10.1155/2019/3682378

Cited by 6 publications

(4 citation statements)

References 10 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…7 A negative Toxoplasma serostatus prior to transplantation has been found to be the only independent risk factor of toxoplasmosis in SOT patients, 6 because both sensitivity (58%-83%) and specificity (60%) of bone marrow aspiration are questionable. 12 Our search strategy allowed us to retrieve 125 cases in SOT recipients, secondary to a variety of causes [66][67][68][69][70][71][72][73][74][75][76][77] (Figure 2). Results are presented in Tables 1 and 2.…”

Section: Discussionmentioning

confidence: 99%

Hemophagocytic syndrome triggered by donor‐transmitted toxoplasmosis as a complication in same‐donor recipients of renal transplantation: Case report and review of the literature

Franci

Flores

Candel

et al. 2021

Transplant Infectious Dis

View full text Add to dashboard Cite

Background Hemophagocytic syndrome (HPS) is an infrequent complication of transplantation caused by an inflammatory response with a benign proliferation of macrophages and defective lytic capability of T lymphocytes and NK cells that can lead to multiorgan failure. Transplant patients are particularly exposed as a result of the increased risk of both infections and malignancies derived from immunosuppressive drugs. There is no consensus for therapy or immunosuppression; mortality is high. We report a case and present a review of all cases of HPS occurring in solid organ transplant recipients. Case report We report two cases of infection by Toxoplasma gondii transmitted by the kidney allograft. One of the recipients was seronegative before transplantation and developed disseminated primary toxoplasmosis. An immune reaction compatible with an HPS ensued. Both were treated with Trimethoprim/sulfamethoxazole, immunosuppression was tapered, and after a 2‐week period a complete response was obtained. Conclusion HPS presents therapeutic challenges in the context of transplantation. If HPS is suspected, the search of a very likely underlying infection should be central to the management.

show abstract

Section: Discussionmentioning

confidence: 99%

Hemophagocytic syndrome triggered by donor‐transmitted toxoplasmosis as a complication in same‐donor recipients of renal transplantation: Case report and review of the literature

Franci

Flores

Candel

et al. 2021

Transplant Infectious Dis

View full text Add to dashboard Cite

show abstract

“…In comparison, our patient developed sHLH 3.5 years after the surgery. There have been only a few documented cases of post-kidney transplant patient developing sHLH late after the transplantation [ 6 , 8 , 9 ]. Specifying the exact pathophysiology of sHLH in our case poses a great challenge.…”

Section: Discussionmentioning

confidence: 99%

“…We present the development and clinical course of sHLH in a kidney transplant recipient. The literature on the subject has been scarce [ 5 , 6 , 7 , 8 ], with the biggest series involving 17 cases of sHLH among a cohort of 4,230 kidney graft recipients [ 9 ]. Our case was complicated by the pulmonary lesion different from the typical complications observed in HLH.…”

Section: Introductionmentioning

confidence: 99%

Secondary Hemophagocytic Lymphohistiocytosis Complicated by a Cavitary Lung Lesion in a Kidney Transplant Recipient

Muras-Szwedziak

Tylski

Masajtis-Zagajewska

et al. 2021

Case Rep Nephrol Dial

View full text Add to dashboard Cite

Hemophagocytic lymphohistiocytosis (HLH) is an uncommon life-threatening condition caused by an uncontrolled immunological response. It can develop secondary to malignancies, infections, systemic diseases, and immunosuppression. Multiple risk factors may present in kidney transplant recipients; however, the cases of HLH in this population have been described sparsely. We report a case of a 39-year-old female kidney transplant recipient who presented to the hospital nearly 3.5 years after the transplantation with general malaise, recent history of weight loss, fevers, and persistent anemia. Laboratory tests showed pancytopenia, hyperferritinemia, hypertriglyceridemia, and increased activity of lactate dehydrogenase. A bone marrow aspiration revealed hemophagocytosis, which led to the diagnosis of HLH. Therapy consisting of high-dose steroids and plasma exchanges was administered, resulting in a significant improvement of blood count parameters and the patient’s general condition. While searching for the triggering disease, a single cavitary lesion in the right lung was revealed in a chest radiograph. Computed tomography scan, bronchoscopy, and additional laboratory testing did not reveal a definitive cause of the lesion. We suspect that the lesion may be a consequence of HLH. The patient was disqualified from thoracic surgery due to multiple comorbidities. Even though HLH is a rare condition, it should be taken into consideration in a kidney transplant patient presenting with unspecific symptoms accompanied by a bicytopenia. It has an unpredictable course that often results in serious complications. Thus close follow-up of the patient and a wide array of imaging and laboratory tests remain crucial.

show abstract

“…Cytomegalovirus infection is the single most frequent cause of infectious morbidity and mortality in kidney transplant recipients. 7 8 However, cytomegalovirus-associated HPS has rarely been described in kidney transplant recipients since the first report of reactive HPS in kidney transplant recipient by Risdall, et al 9 10 11 12 13 14 15 16 In this rare disease, hepatic dysfunction is often present, but liver biopsy has not been performed on the suspicion of HPS. Here, we report a rare case of cytomegalovirus-induced hepatitis combined with reactive HPS in a kidney transplant recipient that was confirmed by hepatic histology.…”

Section: Introductionmentioning

confidence: 99%

Cytomegalovirus-Associated Hemophagocytic Syndrome Diagnosed by Liver Biopsy in a Kidney Transplant Recipient

Choi

Lee

et al. 2021

Yonsei Med J

View full text Add to dashboard Cite

Hemophagocytic syndrome (HPS) is a rare but potentially life-threatening disease in kidney transplant recipients, and is caused by systemic proliferation of macrophages actively phagocytizing other blood cells in the bone marrow, lymph nodes, and the spleen. Here, we report a 40-year-old male kidney transplant recipient who presented with fever, bicytopenia, and elevated liver enzymes 2 months after transplantation. Given that cytomegalovirus antigenemia and real-time polymerase chain reaction tests were positive, liver biopsy was performed under an assumption of cytomegalovirus-induced hepatitis. Hepatic histology revealed multifocal microabscess with cytomegalovirus inclusion bodies, marked Kupffer cell hyperplasia, and erythrophagocytosis by activated macrophages. As laboratory findings such as hyperferritinemia, elevated serum lactate dehydrogenase, low natural killer cell activity, and high soluble interleukin-2 receptor were also compatible with HPS, the recipient was diagnosed as having cytomegalovirus-induced hepatitis combined with reactive HPS. Following intravenous ganciclovir therapy with continuous administration of tacrolimus and corticosteroid, the symptoms resolved and laboratory findings were normalized. As far as we know, this is the first report of cytomegalovirus-induced hepatitis combined with reactive HPS in a kidney transplant recipient that is diagnosed by liver biopsy.

show abstract

Unusual Presentation of Hemophagocytic Lymphohistiocytosis in a Kidney Transplant Patient

Cited by 6 publications

References 10 publications

Hemophagocytic syndrome triggered by donor‐transmitted toxoplasmosis as a complication in same‐donor recipients of renal transplantation: Case report and review of the literature

Hemophagocytic syndrome triggered by donor‐transmitted toxoplasmosis as a complication in same‐donor recipients of renal transplantation: Case report and review of the literature

Secondary Hemophagocytic Lymphohistiocytosis Complicated by a Cavitary Lung Lesion in a Kidney Transplant Recipient

Cytomegalovirus-Associated Hemophagocytic Syndrome Diagnosed by Liver Biopsy in a Kidney Transplant Recipient

Contact Info

Product

Resources

About